Ochs Hans D, Notarangelo Luigi D
Department of Pediatrics, University of Washington, Children's Hospital and Regional Medical Center, 4800 Sand Point Way NE, Seattle, WA 98105, USA.
Curr Allergy Asthma Rep. 2004 Sep;4(5):339-48. doi: 10.1007/s11882-004-0082-5.
Recent advances in molecular genetics have allowed identification of at least seven genes involved in X-linked immunodeficiencies. This has resulted not only in improved diagnostic possibilities but also in a better understanding of the pathophysiology of these disorders. In some cases, mutations in the same gene have been shown to cause distinct clinical and immunologic phenotypes, demonstrating a strong genotype-phenotype correlation. Identification of the molecular basis of these diseases has permitted creation of disease-specific registries, with a better characterization of the clinical and immunologic features associated with the various forms of X-linked immunodeficiencies. Additionally, gene therapy has been attempted in X-linked severe combined immune deficiency (XSCID), with clear evidence of successful correction of the pathology, and the appearance of severe adverse effects.
分子遗传学的最新进展已使至少七种与X连锁免疫缺陷相关的基因得以鉴定。这不仅带来了更好的诊断可能性,还增进了对这些疾病病理生理学的理解。在某些情况下,同一基因的突变已被证明可导致不同的临床和免疫表型,显示出强烈的基因型-表型相关性。这些疾病分子基础的鉴定使得能够建立疾病特异性登记库,从而更好地描述与各种形式X连锁免疫缺陷相关的临床和免疫特征。此外,已尝试对X连锁严重联合免疫缺陷(XSCID)进行基因治疗,有明确证据表明病理得到成功纠正,但也出现了严重的不良反应。